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RESEARCH ARTICLE
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Abstract
Background: Obstetric fistula is a severe condition which has devastating consequences for a womans life. The
estimation of the burden of fistula at the population level has been impaired by the rarity of diagnosis and the lack
of rigorous studies. This study was conducted to determine the prevalence and incidence of fistula in low and
middle income countries.
Methods: Six databases were searched, involving two separate searches: one on fistula specifically and one on
broader maternal and reproductive morbidities. Studies including estimates of incidence and prevalence of fistula at
the population level were included. We conducted meta-analyses of prevalence of fistula among women of
reproductive age and the incidence of fistula among recently pregnant women.
Results: Nineteen studies were included in this review. The pooled prevalence in population-based studies was
0.29 (95% CI 0.00, 1.07) fistula per 1000 women of reproductive age in all regions. Separated by region we found
1.57 (95% CI 1.16, 2.06) in sub Saharan Africa and South Asia, 1.60 (95% CI 1.16, 2.10) per 1000 women of reproductive
age in sub Saharan Africa and 1.20 (95% CI 0.10, 3.54) per 1000 in South Asia. The pooled incidence was 0.09 (95%
CI 0.01, 0.25) per 1000 recently pregnant women.
Conclusions: Our study is the most comprehensive study of the burden of fistula to date. Our findings suggest that
the prevalence of fistula is lower than previously reported. The low burden of fistula should not detract from their
public health importance, however, given the preventability of the condition, and the devastating consequences of
fistula.
Keywords: Vesicovaginal fistula, Maternal morbidity, Systematic review
Background
The World Health Organisation defines an obstetric fistula (referred to as fistula in the text below) as an abnormal opening between a womans vagina and bladder
and/or rectum through which her urine and/or faeces
continually leak [1]. Classifications of fistula vary, but
they generally include fistulae from obstetric causes including vesicovaginal fistula (VVF) and rectovaginal fistula (RVF). Fistulae have devastating consequences [2,3],
particularly in low income countries where women have
less geographical and financial access to appropriate surgical care for repair. In high income countries they are
also devastating, but they are very rare and surgery to
repair them occurs more rapidly.
* Correspondence: alma.adler@lshtm.ac.uk
London School of Hygiene & Tropical Medicine, Keppel St, London, WC1E
7HT, UK
2013 Adler et al.; licensee BioMed Central Ltd. This is an open access article distributed under the terms of the Creative
Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and
reproduction in any medium, provided the original work is properly cited.
Page 2 of 14
series show high rates of divorce or separation [7,8], absence of sexual intercourse [6,7], loss of fertility and
amenorrhea [9,10] and depression [8,11] among women
who have a fistula.
Fistulae are thought to have the highest prevalence
where maternal mortality is high, but there is great uncertainty about the actual prevalence [2]. In the 2000
Global Burden of Disease, Dolea and AbouZhar estimated that 0.08% of all births and 2.15% of neglected
obstructed labour births resulted in fistula [12]. These
estimates came from four studies only, all of which were
in sub-Saharan Africa and two were hospital-based. In
2006, the WHO estimated that more than 2 million
young women throughout the world live with untreated
fistula, and that between 50,000 and 100,000 new
women are affected each year [1]. These statistics originated from countries rapid needs assessments and physicians reports, mostly available in the grey literature,
and not from epidemiological studies using robust design, and almost none include a denominator.
In 2007, Stanton and colleagues wrote a paper [13] on
the challenges of quantifying fistula. They described
three types of publications reporting on frequency, incidence or prevalence of fistula. The first category of papers relied on secondary and tertiary citations (many of
which culminated in personal communications) and reported the number of patients treated without denominators. The second type of publications relied on
No
Exclude
Methods
A two-stage systematic review was conducted in accordance with the STROBE guidelines (http://www.strobestatement.org/) using free-text and subject headings in
Pubmed, Embase, Popline, Lilacs, WHOs Eastern Mediterranean database, and African Index Medicus published
until the end of 2012. The first stage targeted studies specifically reporting on fistula in the title, abstract or subject
headings, including search terms such as fistula, vesicovaginal fistula, and VVF. The second stage aimed at identifying additional studies that examined postpartum and
reproductive morbidity more broadly, whether or not fistula was specifically mentioned as a pathology. This search
Clear
denominator and
adequate follow
up?
Yes
Include
declarations made by the authors themselves, or on surgeons estimates but the source of data was unclear.
The third type of studies described methods and provided appropriate denominators, but with varying degrees of transparency. Stanton and colleagues were only
able to find four papers in this third category [13].
The aim of our review is to provide improved estimates
of the prevalence and incidence of fistula by broadening
the search and including studies that may previously have
been overlooked. Unlike previous reviews of fistula
[12-14], we include studies that examine a broad spectrum
of reproductive morbidity (including morbidity studies
where fistula is but one of many outcomes studied) and/or
where no cases of fistula were reported.
Fistula studies
Reproductive
morbidity studies
Clear
denominator?
Include fistula
as morbidity?
Yes
Include
No
Exclude
Yes
Have
Physician
Exam
Yes
Include
No
No
Exclude
Yes
Include
No
Exclude
included terms such as reproductive morbidity and maternal morbidity. A full search strategy is available upon request. Only English terms were used in the search, but
articles were not excluded based on language.
Reference lists were searched for additional articles.
Using Web of Science, all relevant articles were subjected
to forward citation searching to obtain further articles.
As illustrated in Figure 1, the following study designs
were included: (i) cross-sectional or cohort studies of fistula with hospital based recruitment of pregnant or recently delivered women where the women were followed
for and examined at least 30 days after the end of pregnancy and there was a clear denominator; (ii) cross sectional or cohort studies of prevalent or incident cases of
fistula in the community and (iii) cross sectional or cohort studies of prevalent or incident cases of reproductive morbidity in the community. Studies were only
included if women were examined for the presence of
fistula in hospital and/or if a well trained provider performed a physical examination of the genital area. Morbidity studies not reporting or mentioning fistulae but
including a robust design, a thorough physical exam of
the genital area that reported cases of uterine prolapse
were assumed to have zero cases of fistula, as it was assumed that if fistula had been found it would have been
reported. Studies relying on womens self-reports were
excluded because self-reports of reproductive morbidity
have been shown to be unreliable [13,15,16]. We excluded studies that were conducted before 1990 or published before 1991. We included studies irrespective of
sample size but studies without a denominator were excluded. If more than one paper provided results of the
same study population, data were first extracted from
the article with the greatest amount of information,
and supplementary data extracted from the other papers if required.
Data were extracted by a single author (AJA) using a
proforma and included information on region, study dates,
study population, duration of fistula, type of fistula, risk
factors, associated complications and denominators,
how fistula were ascertained, sampling technique, number of women and number of deliveries. There are no
good tools for looking at study quality in crosssectional studies, so study quality was assessed using a
modified Ottawa-Newcastle score (http://www.ohri.ca/
programs/clinical_epidemiology/oxford.asp).
The studies reported on two types of populations of
women: women of reproductive age and women with a recent pregnancy. In studies targeting women of reproductive
age we calculated the prevalence of fistula per 1000 women
of reproductive age. In studies where women were followed
after end of pregnancy, we calculated the incidence of fistula per 1000 recently pregnant women (assuming that
women were unlikely to have had fistula before getting
Page 3 of 14
pregnant). In all cases 95% confidence intervals were calculated using the binomial exact method.
Meta-analyses were conducted using the metaprop
command from the R 2.15.3 package meta using a
random-effects model [17]. Meta-analyses were conducted
summarising the prevalence of fistula among women of
reproductive age and the incidence of fistula in recently
pregnant women. All studies were stratified by continent
and by recruitment in hospital or in the community.
Results
The initial search of studies focussing on fistula found
9130 references after de-duplication, 367 of which were
retained after title and abstract screening. From the 367
articles, six were found to have information on either
prevalence or incidence of fistula [18-23]. A further 13
After removing
duplicates 9130
Found from
general maternal
and reproductive
morbidity studies
10
Found from
forward
searching and
reference lists 10
Final: 19 Studies
Author
Study design
Assessment of fistula
Number of fistula
Number of
women/
pregnancies
Prevalence
(per 1000 WRA)
Cross-sectional survey of
obstetric fistula
44 (untreated)
19,153
1,038
1,167
385
509
506
Oman, Mixed
1,662
379
696
Comprehensive gynaecological
examination of all married
women including a speculum
examination [1]
1117
Page 4 of 14
Study area
Repair
34
32,188
Repair
111
425,865
Repair
66
150,000
[1] These studies were reproductive morbidity studies which did not state in the methods that they were investigating fistula, nor did they report any cases of fistula; however the type of examination used to identify
other reproductive morbidities was assessed to have been sufficient that should there have been any cases of fistula they would have been identified.
Table 1 Characteristics of studies reporting fistula prevalence included in the review (Continued)
Page 5 of 14
Author
Study area
Study design
Assessment of fistula
Number of
fistula
Number of women/
pregnancies
Vangeenderhuysen et al.,
2001 [22]
19,694
1,162
0 (0, 3.17)
557
0 (0.0. 6.6)
Medical examinations
1,014
709
Medical examinations
4,081
Page 6 of 14
Page 7 of 14
studies [24-34] were found from searching general maternal and reproductive morbidity studies (including
three from reference lists and forward citation searching
[34-36]) (Figure 2).
Of the 19 studies, 13 were community-based; seven of
which reported on fistula [18-20,22,24,33,35] and six
did not mention fistula anywhere in the paper. Ten
community-based studies reported the prevalence of fistula among women of reproductive age (Table 1) and
three reported the incidence among recently pregnant
women (Table 2). Six studies recruited women in hospital and followed them for 30 days or more after the
end of pregnancy (Table 2). Three studies [21,23,36], report the prevalence of fistula among women after having
fistula repairs. These studies were included because it
was possible to understand the population that these
women came from, and have a denominator (Table 1).
The other three studies reported the incidence of fistula
among women after giving birth or miscarrying in hospital. These studies recruited women with obstetric
complications as well as a sample of women without
complications [30-32].
Figure 4 Prevalence of fistula per 1000 women of reproductive age stratified by region.
Page 8 of 14
Figure 5 Prevalence of fistula per 1000 women of reproductive age in studies with hospital-based recruitment.
Africa had zero events, it is impossible for the metaanalysis to provide a meaningful estimate due to the manner with which zero prevalence studies are dealt with
(http://cran.r-project.org/web/packages/meta/meta.pdf).
The prevalence of fistula in studies with hospital-based
recruitment ranged from 0.26 to 1.06 per 1000 women
of reproductive age, with a median of 0.44 per 1000
women of reproductive age. The pooled estimate of the
prevalence of fistula in hospital was 0.51 (three studies
with 608,053 participants 95% CI 0.25, 0.87) per 1000
women of reproductive age (Figure 5).
The pooled incidence of fistula in community-based
studies was 0.09 (three studies with 21,413 participants
0.01, 0.25) per 1000 recently pregnant women (Figure 6)
and in hospital-based studies 0.66 (three studies including 5804 participants 0.16, 1.48) per 1000 recently pregnant women (Figure 7).
The I [2] values varied substantially by stratum, ranging
from 0% in community-based (Figure 6) and hospitalbased (Figure 7) incidence studies to 94.8% in prevalence
studies with hospital based recruitment (Figure 5). Study
quality in the modified Ottawa-Newcastle score table is
shown in Table 3. All studies had a cross-sectional or cohort design.
Only two studies reported information on duration of
fistula: one found a median of eight years [18], and the
Discussion
Our comprehensive systematic review found that fewer
than 1 per 1000 women of reproductive age in low and
middle income countries suffer from fistula; this figure
rises to 1.57 per 1000 when only data from sub-Saharan
Africa and South Asia is used. The number of new cases
of fistula ranged from 0.09 per 1000 recently pregnant
women in community-based studies to 0.66 per 1000
pregnancies in hospital-based studies.
The WHO has suggested that over two million women,
mostly from sub-Saharan African and Asian countries,
have fistula [1,37]. Given an estimated population of 645
million women of reproductive age in sub-Saharan Africa
and South Asia in 2010 (http://esa.un.org/wpp/unpp/
p2k0data.asp), this would suggest that 3 per 1000 women
of reproductive age have a fistula, which is considerably
higher than our estimate for low and middle income
countries. There were too few studies in our review to arrive at robust estimates of the prevalence of fistula by continent, but even the highest estimates for sub-Saharan
Africa (1.62 per 1000 women of reproductive age in
Ethiopia) or South Asia (2.6 per 1000 in India) fall well
Figure 6 Incidence of fistula per 1000 pregnancies in community-based studies, stratified by region.
Page 9 of 14
Figure 7 Incidence of fistula per 1000 pregnant women in studies with hospital-based recruitment.
Selection
Comparability Outcome
Study:
Representativeness of
the study population
Comparability Assessment
of cases and
of outcome
non-cases
Population based
sample of seven
administrative
regions of rural
Ethiopia
Do not state
Walraven et al.,
2001 [24]
Obstetric morbidities
including fistula
Population based
rural region
Obstetric morbidities
including fistula
Population based
Gynaecological and
related morbidities, but
do not state that they
looked for fistula
Population based
No cases
Do not state
Population based
No cases
Gynaecological and
related morbidities, but
do not state that they
looked for fistula
Population based
from national
survey
No cases
Gynaecological and
related morbidities, but
do not state that they
looked for fistula
Population based
No cases
Do not state
Page 10 of 14
No cases
Gynaecological and
related morbidities,
but do not state
that they looked for
fistula
Population based
No cases
Obstetric fistula
No non cases
Physical exam
and treatment
N/A
Kalilani-Phiri et al.,
2010 [21]
Obstetric fistula
No non cases
Physical exam
and treatment
N/A
Obstetric fistula
No non cases
Physical exam
and treatment
N/A
Vangeenderhuysen
et al., 2001 [22]
Obstetric morbidities
including fistula
Population based
Obstetric morbidities
including fistula
Population based
Severe obstetric
complications
including fistula
Women with
complications overrepresented but also
had follow up of women
with uncomplicated birth
Severe obstetric
complications
including fistula
Women with
complications overrepresented but also
had follow up of women
with uncomplicated birth
Severe obstetric
complications
including fistula
Page 12 of 14
Page 13 of 14
Conclusions
Our study is the most comprehensive study of the burden of fistula to date, including study sources not generally used. Our findings suggest that the prevalence and
incidence of fistula is relatively low. The low burden of
fistula should not detract from their public health importance, however, given the preventability of the condition, and the devastating consequences of fistula. Future
studies of fistula should include a description of the
study population with defined denominators.
Competing interests
The authors declare that they have no competing interests.
Authors contributions
AJA conducted search, extracted data, did analysis and wrote first draft of
paper CR advised on methodology and commented on drafts CC
commented on drafts and helped in writing of paper VF helped design
study and commented on paper. All authors read and approved the final
manuscript.
Acknowledgements
The authors would like to thank Doris Chou, Lale Say, and Herbert Peterson
for their comments.
Funding
This research was funded through a grant made to the Child Health
Epidemiology Reference Group (CHERG) by the Bill and Melinda Gates
Foundation.
Received: 13 June 2013 Accepted: 9 December 2013
Published: 30 December 2013
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